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					    Provision of dental implants in
patients diagnosed with scleroderma.

     A rapid systematic review.




                          By

      WorkSafeBC Evidence-Based Practice Group

     Dr. Craig W. Martin, Senior Medical Advisor

                       April 2009




            Clinical Services - Worker and Employer Services
   Provision of dental implants in patients diagnosed with scleroderma.
                         A rapid systematic review.


Background

         In early November 2008, the Evidence Based Practice Group (EBPG) was asked if the
Board should provide dental implants to an injured worker diagnosed with scleroderma (of
systemic type). Given this question, the EBPG conducted a rapid systematic review in order to
look for evidence on:

    what disease characteristics of scleroderma may hinder dental implantation

    what the survival length of dental implants is among patients with scleroderma

    what alternative treatments are available for partial or complete edentulous scleroderma
    patients




Methods

    A systematic literature search was conducted on November 12, 2008.

    The search was done on commercial medical literature databases, including MEDLINE,
    MEDLINE Daily Update, EMBASE and BIOSIS Previews. These databases are available
    through the Ovid SP interface.

    The search employed the keywords:

         ((scleroderma or raynaud) and (implant or dental implant or prosthesis or dental
         prosthesis or dental or dentistry))

    Searches were limited to studies conducted in human subjects as well as to studies
    published in the English language (or where at least the abstract was in English). There was
    no limitation on study design put in place. Further, a manual search was also conducted on
    review articles in order to identify studies that might provide data to answer the objectives
    of this rapid review.

    The search yielded 444(1-444) published articles. A further four articles(445-448) were retrieved
    as the result of manual searching. Upon examination of the titles and abstracts of these
    articles, 29(12,67,92,93,111,116,147,174,200,205,246,260,270,285,296,316,320,328,332,341,348,365,375,399,436,445-448) were
    retrieved in full for further appraisal. A textbook on rheumatology(449,450) was also consulted.



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Provision of dental implants in patients diagnosed with scleroderma                                     2


     Of these 29 articles, four(348,316,285,93) were excluded due to irrelevancy.

     A level of evidence (Appendix 1) was assigned for each article according to the levels of
     evidence adopted by the EBPG.




Results

         Scleroderma, which originates from the Greek words “scleros” meaning hard and
“derma” meaning skin, is thought to be an autoimmune multisystem rheumatic condition that
affects connective tissues.(12,67) Clinically, scleroderma is characterized by thickening and
induration with diffuse fibrosis of the skin as well as various internal organs.(67,111) Hardening of
the skin (i.e. scleroderma) is usually the initial manifestation of the disease process, which may
be better described as progressive systematic sclerosis. Progressive systemic sclerosis itself can
further be subdivided into systemic sclerosis, which can involve virtually any organ system and
localized sclerosis, which usually is limited to skin without any internal organ involvement.(12,116)

         Progressive systemic sclerosis (PSS) occurs more commonly in women, with a female to
male ratio of 4:1 and peak age of onset at 30 – 50 years of age.(12) It is an uncommon condition
with an estimated prevalence of the disease, in the US, of 400 per million women between the
ages of 35 – 65 years.(12) Other experts estimate the prevalence of PSS to be around 4 to 253
cases per million persons, with an estimated annual incidence of 10 to 20 cases per million
persons.(445) The incidence of the disease is estimated at 4 to 19 new cases/million/year.(116,332)
(Please note that the wide range of incidence and prevalence numbers occur as a result of
differing diagnostic criteria and study populations.)

         Oral manifestations, including trigeminal neuropathy, xerostomia, thickened periodontal
ligament, blunting of mandibular angles on dental radiographs, microstomia, idiopathic
resorption of tooth(92) and bone, pathologic mandibular fracture, and poor oral hygiene, are
experienced commonly by patients.(205,341) Inadequate salivary flow (xerostomia) due to fibrosis
of the salivary duct compromises buffering within the oral cavity and allows the acidity
produced by bacterial metabolism and gastric acid (due to gastro-oesophageal reflux) to erode
dentition (which can be observed as caries as well as periodontal disease.)(332) The thickening of
the periodontal ligament or periodontal ligament space, that may affect all teeth, is also a
common manifestation of PSS.(399) Collagenous changes of the oral mucosal membrane cause it
to appear thin, pale and tight, with ulcerations due to the loss of vascular integrity in the oral
mucosal membrane. This mucosal fibrosis may also induce gingival recession and stripping of
the attached gingiva.(67) Decrease in vascularity and oral tissue ischemia is thought to be the
major cause of observed periodontal disease and mobile teeth.(67,332) Resorption of the mandible
is another common oral manifestation of PSS.(116,320,365,436) Mandibular resorption occurs as a
result of facial skin tightening, vessel constriction, and the underlying taut musculature that
exerts continuous pressure, causing ischemia to the mandible.(166,399,446,447) Even though the


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Provision of dental implants in patients diagnosed with scleroderma                                   3


majority of this bone resorption phenomenon is asymptomatic, the areas of mandibular
resorption are very problematic since it increases the risk of mandibular fracture as well as
trigeminal neuropathy and osteomyelitis.(246,399) It should also be noted that a case of cervical
spine and mandible bone resorption in a patient with PSS has been reported in the literature.(448)
A reduced oral opening (i.e. microstomia), resulting from excess collagen deposition in the skin
and perioral tissues, occurs in about 70%-80% of patients.(205,341) These findings of xerostomia,
microstomia, decreased vascularity (that affects tissue healing), continuous increased oral tissue
pressure due to fibrosis (tissue ischemia), bone resorption, and tooth mobility are directly
relevant to the present and future dental management of patients diagnosed with scleroderma.

         Experts argue that successful dental management of patients with PSS is dependent on
the ability to control the disease, including the need to take into account the anticipated
worsening of the dental condition (level of evidence 5).(12,67,147,399) Given the many oral
manifestations of PSS, including microstomia, xerostomia, caries, gingivitis, oral candidiasis,
mandibular bone resorption, temporomandibular disorders, enamel erosion, dysphagia, and
delayed tissue healing, dental treatment in the PSS patient may require the involvement of all
members of health care team including the internist, rheumatologist, dermatologist, dental
hygienist, oral pathologist, oral surgeon, endodontist, and periodontist, as well as
prosthodontist (level of evidence 5).(67,328,375) Experts argue that in PSS patients, many of the
traditional approaches for dental restoration need to be modified -- for instance, to
accommodate microstomia. Further, although PSS patients, at present, may be able to tolerate
the procedures, as the disease progresses there may be problems in maintenance of the existing
restoration as well as in future replacement.(67)

        Many methods of treating partial or complete edentulous PSS patients have been
reported in the literature.(116,147,200,270,296,328,375) These methods include maxillary and mandibular
complete dentures,(116) implant-retained fixed partial dentures,(147,270) (maxilla or mandible)
overdentures retained by osseointegrated implants(200,296) and a removable partial denture.(328) It
should be noted that all of these examples are case reports (level of evidence 5) with no follow
up results being reported.

         With regard to the provision of implants for dental restoration in PSS patients, Jensen
and Sindet-Pedersen(174) reported on a case of the use of osseointegrated implants for
reconstruction in a PSS patient with multiple external and internal root resorptions (level of
evidence 5). Briefly, Jensen and Sindet-Pedersen(174) reported installing nine mandible implants
(four distal and five mesial to the mental foramina) in a 39-year-old male patient diagnosed with
PSS who had extensive internal and external root resorptions which were most severe in the
mandibular teeth. Four months after the implantation, it was possible to connect abutments on
only six out of the nine implants; one implant was not integrated and the two most distal
implants were not possible to connect to due to microstomia. At 24 months follow up, the
authors reported no pocket formation or other radiographic abnormalities around the implants.
It should be noted that Jensen and Sindet-Pedersen(174) strongly stressed the importance of the



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Provision of dental implants in patients diagnosed with scleroderma                                   4


success in controlling the scleroderma itself, which they thought to be responsible for the
favourable biologic reaction to the implants (c.q. no inflammation or marginal bone destruction)
in this patient. Further, the authors stated that even though the implants remained healthy at
two years follow up, it was impossible to predict the long term outcome of treatment since
reaction to the implants might ultimately develop in the adjacent connective tissue. This
uncertainty in the prospect of dental implants in PSS patients was also stressed by Chaffe.(67)




Summary

     Progressive Systemic Sclerosis (PSS) is an autoimmune multisystem rheumatic condition that
     affects connective tissues of various organs, including orofacial connective tissue.

     Many PSS oral manifestations, including xerostomia, microstomia, caries, decreased
     vascularity (which affects tissue healing), continuous increased oral tissue pressure due to
     fibrosis (tissue ischemia), bone resorption, and tooth mobility may affect the choice and
     survival of dental implants in affected patients. Microstomia, bone resorption, and potential
     adjacent tissue reaction to implants may be detrimental to the provision of implant
     technology in edentulous PSS patients.

     At present there is only one case report on the provision of osseointegrated implants in a
     well controlled (treated) PSS patient (level of evidence 5). It is not possible to predict the
     survival of the tooth implants given the available information.

     Other alternatives to treat edentulous PSS patients, which have been reported in the
     literature, include complete dentures, implant-retained fixed partial dentures, overdentures
     retained by osseointegrated implants, and a removable partial denture (level of evidence 5).
     However, it should be noted that these reports did not provide information on the
     prognosis or follow up of these dental procedures.




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Provision of dental implants in patients diagnosed with scleroderma                                 5


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WorkSafeBC Evidence-Based Practice Group                   April 2009    www.worksafebc.com/evidence
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WorkSafeBC Evidence-Based Practice Group                   April 2009   www.worksafebc.com/evidence
Provision of dental implants in patients diagnosed with scleroderma                                43


Appendix 1

WorkSafeBC - Evidence-Based Practice Group Levels of Evidence (adapted from 1,2,3,4)


               Evidence from at least 1 properly randomized controlled trial (RCT) or systematic review
      1
               of RCTs.

               Evidence from well-designed controlled trials without randomization or systematic
      2
               reviews of observational studies.

               Evidence from well-designed cohort or case-control analytic studies, preferably from
      3
               more than 1 centre or research group.

               Evidence from comparisons between times or places with or without the intervention.
      4
               Dramatic results in uncontrolled experiments could also be included here.

               Opinions of respected authorities, based on clinical experience, descriptive studies or
      5
               reports of expert committees.



References

1. Canadian Task Force on the Periodic Health Examination: The periodic health examination.
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3. Scottish Intercollegiate Guidelines Network (2001). SIGN 50: a guideline developers'
   handbook. SIGN. Edinburgh.
4. Canadian Task Force on Preventive Health Care. New grades for recommendations from the
   Canadian Task Force on Preventive Health Care. CMAJ. Aug 5, 2003;169(3):207-208.




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